The skeletal phenotype of chondroadherin deficient mice.

Chondroadherin, a leucine rich repeat extracellular matrix protein with functions in cell to matrix interactions, binds cells via their α2β1 integrin as well as via cell surface proteoglycans, providing for different sets of signals to the cell. Additionally, the protein acts as an anchor to the mat...

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Autores principales:	Lovisa Hessle, Gunhild A Stordalen, Christina Wenglén, Christiane Petzold, Elizabeth K Tanner, Sverre-Henning Brorson, Espen S Baekkevold, Patrik Önnerfjord, Finn P Reinholt, Dick Heinegård
Formato:	article
Lenguaje:	EN
Publicado:	Public Library of Science (PLoS) 2014
Materias:	Medicine R Science Q
Acceso en línea:	https://doaj.org/article/b9105d4a8e204ba0b64b08144ae7442c
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spelling	oai:doaj.org-article:b9105d4a8e204ba0b64b08144ae7442c2021-11-18T07:43:19ZThe skeletal phenotype of chondroadherin deficient mice.1932-620310.1371/journal.pone.0063080https://doaj.org/article/b9105d4a8e204ba0b64b08144ae7442c2014-01-01T00:00:00Zhttps://www.ncbi.nlm.nih.gov/pmc/articles/pmid/23755099/pdf/?tool=EBIhttps://doaj.org/toc/1932-6203Chondroadherin, a leucine rich repeat extracellular matrix protein with functions in cell to matrix interactions, binds cells via their α2β1 integrin as well as via cell surface proteoglycans, providing for different sets of signals to the cell. Additionally, the protein acts as an anchor to the matrix by binding tightly to collagens type I and II as well as type VI. We generated mice with inactivated chondroadherin gene to provide integrated studies of the role of the protein. The null mice presented distinct phenotypes with affected cartilage as well as bone. At 3-6 weeks of age the epiphyseal growth plate was widened most pronounced in the proliferative zone. The proteome of the femoral head articular cartilage at 4 months of age showed some distinct differences, with increased deposition of cartilage intermediate layer protein 1 and fibronectin in the chondroadherin deficient mice, more pronounced in the female. Other proteins show decreased levels in the deficient mice, particularly pronounced for matrilin-1, thrombospondin-1 and notably the members of the α1-antitrypsin family of proteinase inhibitors as well as for a member of the bone morphogenetic protein growth factor family. Thus, cartilage homeostasis is distinctly altered. The bone phenotype was expressed in several ways. The number of bone sialoprotein mRNA expressing cells in the proximal tibial metaphysic was decreased and the osteoid surface was increased possibly indicating a change in mineral metabolism. Micro-CT revealed lower cortical thickness and increased structure model index, i.e. the amount of plates and rods composing the bone trabeculas. The structural changes were paralleled by loss of function, where the null mice showed lower femoral neck failure load and tibial strength during mechanical testing at 4 months of age. The skeletal phenotype points at a role for chondroadherin in both bone and cartilage homeostasis, however, without leading to altered longitudinal growth.Lovisa HessleGunhild A StordalenChristina WenglénChristiane PetzoldElizabeth K TannerSverre-Henning BrorsonEspen S BaekkevoldPatrik ÖnnerfjordFinn P ReinholtDick HeinegårdPublic Library of Science (PLoS)articleMedicineRScienceQENPLoS ONE, Vol 8, Iss 6, p e63080 (2014)
institution	DOAJ
collection	DOAJ
language	EN
topic	Medicine R Science Q
spellingShingle	Medicine R Science Q Lovisa Hessle Gunhild A Stordalen Christina Wenglén Christiane Petzold Elizabeth K Tanner Sverre-Henning Brorson Espen S Baekkevold Patrik Önnerfjord Finn P Reinholt Dick Heinegård The skeletal phenotype of chondroadherin deficient mice.
description	Chondroadherin, a leucine rich repeat extracellular matrix protein with functions in cell to matrix interactions, binds cells via their α2β1 integrin as well as via cell surface proteoglycans, providing for different sets of signals to the cell. Additionally, the protein acts as an anchor to the matrix by binding tightly to collagens type I and II as well as type VI. We generated mice with inactivated chondroadherin gene to provide integrated studies of the role of the protein. The null mice presented distinct phenotypes with affected cartilage as well as bone. At 3-6 weeks of age the epiphyseal growth plate was widened most pronounced in the proliferative zone. The proteome of the femoral head articular cartilage at 4 months of age showed some distinct differences, with increased deposition of cartilage intermediate layer protein 1 and fibronectin in the chondroadherin deficient mice, more pronounced in the female. Other proteins show decreased levels in the deficient mice, particularly pronounced for matrilin-1, thrombospondin-1 and notably the members of the α1-antitrypsin family of proteinase inhibitors as well as for a member of the bone morphogenetic protein growth factor family. Thus, cartilage homeostasis is distinctly altered. The bone phenotype was expressed in several ways. The number of bone sialoprotein mRNA expressing cells in the proximal tibial metaphysic was decreased and the osteoid surface was increased possibly indicating a change in mineral metabolism. Micro-CT revealed lower cortical thickness and increased structure model index, i.e. the amount of plates and rods composing the bone trabeculas. The structural changes were paralleled by loss of function, where the null mice showed lower femoral neck failure load and tibial strength during mechanical testing at 4 months of age. The skeletal phenotype points at a role for chondroadherin in both bone and cartilage homeostasis, however, without leading to altered longitudinal growth.
format	article
author	Lovisa Hessle Gunhild A Stordalen Christina Wenglén Christiane Petzold Elizabeth K Tanner Sverre-Henning Brorson Espen S Baekkevold Patrik Önnerfjord Finn P Reinholt Dick Heinegård
author_facet	Lovisa Hessle Gunhild A Stordalen Christina Wenglén Christiane Petzold Elizabeth K Tanner Sverre-Henning Brorson Espen S Baekkevold Patrik Önnerfjord Finn P Reinholt Dick Heinegård
author_sort	Lovisa Hessle
title	The skeletal phenotype of chondroadherin deficient mice.
title_short	The skeletal phenotype of chondroadherin deficient mice.
title_full	The skeletal phenotype of chondroadherin deficient mice.
title_fullStr	The skeletal phenotype of chondroadherin deficient mice.
title_full_unstemmed	The skeletal phenotype of chondroadherin deficient mice.
title_sort	skeletal phenotype of chondroadherin deficient mice.
publisher	Public Library of Science (PLoS)
publishDate	2014
url	https://doaj.org/article/b9105d4a8e204ba0b64b08144ae7442c
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The skeletal phenotype of chondroadherin deficient mice.

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